scholarly journals Improvement of Arterial Wall Lesions in Parallel with Decrease of Plasma Pentraxin-3 Levels in a Patient with Refractory Takayasu Arteritis after Treatment with Tocilizumab

2017 ◽  
Vol 2017 ◽  
pp. 1-5
Author(s):  
Shiho Iwagaitsu ◽  
Taio Naniwa

A 19-year-old Japanese woman with active Takayasu arteritis despite multiple conventional immunosuppressive therapies with glucocorticoids in combination with intravenous cyclophosphamide, azathioprine, or infliximab with methotrexate and tacrolimus was successfully treated by switching from infliximab to intravenous tocilizumab. Worsening of claudication of the legs and elevated acute phase reactants, including plasma pentraxin-3 levels, were observed during combination therapy with infliximab. Computed tomography demonstrated increased wall thickening with contrast enhancement in the preexisting lesion of the descending aorta and the femoral arteries. After switching from infliximab to tocilizumab, plasma pentraxin-3 levels gradually decreased to the normal range in parallel with the improvement of claudication. Follow-up computed tomographic scans confirmed the marked improvement of these arterial lesions. Moreover, plasma pentraxin-3 level was increased in response to the worsening of claudication that occurred just after switching to a subcutaneous tocilizumab injection. Measurements of plasma pentraxin-3 might be useful for evaluation of the vascular wall inflammation and therapeutic efficacy even during biologic therapy targeting tumor necrosis factor α and interleukin-6.

2016 ◽  
Vol 2016 ◽  
pp. 1-5
Author(s):  
Takashi Suzuki ◽  
Satoru Nakayama ◽  
Haruka Muto ◽  
Shinichi Shimizu ◽  
Yoshiro Otsuki ◽  
...  

Here, we report an extremely rare case of trichoepithelioma (TE)—a benign epithelial tumor originating from the outer root sheath of a hair follicle—arising in an ovarian mature cystic teratoma (MCT) with fluorodeoxyglucose-positron emission tomography (FDG-PET) findings. A 48-year-old Japanese woman presented to our hospital for her annual follow-up of adenomyosis. Ultrasonography and magnetic resonance imaging revealed a left ovarian tumor with irregular-shaped septum, which was suspicious of malignancy. However, tumor marker levels were within normal range. On FDG-PET, the maximum standardized uptake value (SUVmax) of the tumor was 2.9. Laparotomy with left salpingooophorectomy was performed. Pathologic examination revealed the probability of TE, rather than basal cell carcinoma (BCC), arising in an ovarian MCT. After five years of follow-up, the patient had no sign of recurrence. The FDG-PET SUVmax was low in TE, as with other benign tumor. However, future investigation is needed to evaluate the findings of FDG-PET imaging in TE cases.


2017 ◽  
Vol 7 (2) ◽  
pp. 112-120 ◽  
Author(s):  
Hitoshi Kodera ◽  
Yasuhide Mizutani ◽  
Satoshi Sugiyama ◽  
Toshio Miyata ◽  
Takashi Ehara ◽  
...  

Lipoprotein glomerulopathy (LPG) is characterized by the accumulation of lipoprotein thrombi within glomerular capillaries. This rare disorder is associated with various types of mutations in the apolipoprotein E gene (apoE). Herein, we present a case of LPG with a combination of apoE Chicago (Arg147Pro) and apoE (Glu3Lys) mutations. A 51-year-old Japanese woman presented with severe (3+) proteinuria. The initial renal biopsy showed glomerular capillary dilation and occlusion with lipid granules, a specific characteristic of LPG. Phenotype, genotype, and apoE DNA sequence analyses detected 2 mutations as described above within the same allele. Although both mutations had already been reported in 1 case of LPG each, this is the first time that the combination of the 2 mutations was identified in the same case. Familial analysis detected the same mutations in the patient’s mother. However, she has not suffered LPG thus far. In addition, a re-analysis of the previous LPG case with apoE (Glu3Lys) also identified the apoE Chicago mutation, as was observed in our case. Treatment with fenofibrate and irbesartan was initiated, and urinary protein excretion ceased within 1 year; recurrence was not observed after an additional 2 years of follow-up. A second biopsy after 2 years showed great improvement, with lipoprotein thrombi identified only in 2 of 18 glomeruli.


2020 ◽  
Vol 2020 ◽  
pp. 1-7
Author(s):  
Veena Dronamraju ◽  
Navneet Singh ◽  
Justin Poon ◽  
Sachi Shah ◽  
Joseph Gorga ◽  
...  

Bronchiectasis is characterized by permanent destruction of the airways that presents with productive cough, as well as bronchial wall thickening and luminal dilatation on computed tomographic (CT) scan of the chest; it is associated with high mortality. Accumulating data suggests higher rates of bronchiectasis among the HIV-positive population. This case series involves 14 patients with bronchiectasis and HIV followed at two major urban institutions from 1999 to 2018. Demographics, clinical presentation, microbiology, radiographic imaging, and outcomes were collected and compiled. Mean age was 42 years (range 12-77 years). 36% had a CD4 count greater than 500 cells/mm3, 28% had a CD4 count between 200 and 500 cells/mm3, and 36% had AIDS. 43% were treated for Pneumocystis jiroveci pneumonia (PJP) and 50% for Mycobacterium avium complex (MAC) infection. 21% had COPD, 7% had asthma, and 7% had a history of pulmonary aspergillosis. Two patients were followed up by pulmonary services after diagnosis of bronchiectasis on CT. The timeline of the follow-up in these cases was within months and after three years respectively. It is posited that the prevalence of bronchiectasis in HIV patients may be underestimated. Improving recognition and management of bronchiectasis could help diminish rehospitalization rates.


2020 ◽  
Vol 28 (6) ◽  
pp. 683-687
Author(s):  
Waka Iwashita ◽  
Atsushi Kurabayashi ◽  
Chiharu Tanaka ◽  
Seiji Naganuma ◽  
Takanori Kawamura ◽  
...  

Superficial angiomyxoma is a rare, benign, multilobulated cutaneous tumor composed of stellate and spindle cells, a prominent myxoid matrix, and numerous blood vessels. Superficial angiomyxoma may be indistinguishable from cutaneous lesions of the Carney complex, although superficial angiomyxoma can occur independently of the complex. In this article, we present the case of a 39-year-old Japanese woman with a 40 × 30 mm, focally ulcerated, polypoid superficial angiomyxoma on the left nipple without any evidence of Carney complex. The development of superficial angiomyxoma on the nipples in a patient without the Carney complex is extremely rare. Indeed, only 3 cases of superficial angiomyxoma arising on the nipple have been reported to date, and this is the first such report in Japan. In such cases, the majority of superficial angiomyxoma of the nipples develop in premenopausal women. The possibility of superficial angiomyxoma should be considered for all polypoid nipple lesion, particularly in premenopausal women, and complete excision with follow-up observation should be performed.


2003 ◽  
Vol 17 (2) ◽  
pp. 111-113 ◽  
Author(s):  
Henry V Chung ◽  
Alnoor Ramji ◽  
Jennifer E Davis ◽  
Sylvia Chang ◽  
Graham D Reid ◽  
...  

Classically, a diagnosis of systemic lupus erythematosus (SLE) is dependent on renal, rheumatological, cutaneous and neurological target organ damage with supporting serological markers. A previously healthy 26-year-old Japanese woman whose only manifestation of otherwise occult SLE was severe abdominal pain is reported. A computed tomographic scan of the abdomen revealed thickened loops of small bowel, endoscopic findings were nonspecific and jejunal biopsy revealed a nonspecific enteritis. Laboratory studies revealed lymphopenia, hypocomplementemia, a positive antinuclear antibody, a weakly positive anti-Smith and a strongly positive anti-double stranded DNA. There was a prompt symptomatic recovery with immunosuppressive therapy. The authors’ experiences, and a review of the literature suggest that a diagnosis of SLE should be considered in young Asian women who present with significant but clinically enigmatic gastrointestinal illness.


2015 ◽  
Vol 6 (1) ◽  
pp. 127-131 ◽  
Author(s):  
Atsuko Sato ◽  
Nami Senda ◽  
Emi Fukui ◽  
Kouichi Ohta

Purpose: To report the presence of retinal angiomatous proliferation (RAP) in an eye with cuticular drusen detected by fluorescein and indocyanine green angiography by confocal scanning laser ophthalmoscopy and by spectral-domain optical coherence tomography (SD-OCT). Methods: Case report of a 65-year-old Japanese woman with cuticular drusen. Results: At her first ophthalmic examination, her visual acuity was 20/20 in both eyes. An ophthalmoscopy showed many small subretinal pigment epithelial deposits in both eyes. These deposits had a ‘saw-tooth pattern' in the SD-OCT images. During the follow-up examination, retinal hemorrhages were observed, and fluorescein angiography showed a ‘stars-in-the-sky' appearance and intraretinal neovascularization. The patient was diagnosed with cuticular drusen associated with RAP. Conclusion: We suggest that the cuticular drusen were associated with RAP, so periodic follow-up examinations are needed for patients with cuticular drusen for the early detection and treatment of RAP.


1996 ◽  
Vol 35 (05) ◽  
pp. 146-152 ◽  
Author(s):  
A. Kögler ◽  
H.-A. Schmitt ◽  
D. Emrich ◽  
H. Kreuzer ◽  
D. L. Munz ◽  
...  

SummaryThis prospective study assessed myocardial viability in 30 patients with coronary heart disease and persistent defects despite reinjection on TI-201 single-photon computed tomography (SPECT). In each patient, three observers graded TI-201 uptake in 7 left ventricular wall segments. Gradient-echo magnetic resonance imaging in the region of the persistent defect generated 12 to 16 short axis views representing a cardiac cycle. A total of 120 segments were analyzed. Mean end-diastolic wall thickness and systolic wall thickening (± SD) was 11.5 ± 2.7 mm and 5.8 ± 3.9 mm in 48 segments with normal TI-201 uptake, 10.1 ± 3.4 mm and 3.7 ± 3.1 mm in 31 with reversible lesions, 11.3 ± 2.8 mm and 3.3 ± 1.9 mm in 10 with mild persistent defects, 9.2 ± 2.9 mm and 3.2 ±2.2 mm in 15 with moderate persistent defects, 5.8 ± 1.7 mm and 1.3 ± 1.4 mm in 16 with severe persistent defects, respectively. Significant differences in mean end-diastolic wall thickness (p <0.0005) and systolic wall thickening (p <0.005) were found only between segments with severe persistent defects and all other groups, but not among the other groups. On follow-up in 11 patients after revascularization, 6 segments with mild-to-moderate persistent defects showed improvement in mean systolic wall thickening that was not seen in 6 other segments with severe persistent defects. These data indicate that most myocardial segments with mild and moderate persistent TI-201 defects after reinjection still contain viable tissue. Segments with severe persistent defects, however, represent predominantly nonviable myocardium without contractile function.


1970 ◽  
Vol 5 (4) ◽  
pp. 61-66
Author(s):  
Patrícia Resende Penido ◽  
Rhanna Junqueira Westin de Carvalho ◽  
Roger Willian Moraes Mendes

RESUMOIntrodução: A Arterite de Takayasu (AT) consiste em uma vasculopatia de origem indefinida, sendo de caráter crônico, que afeta a aorta e seus ramos principais. Em gestantes é uma condição complexa, em que o tratamento clínico é realizado com restrições. A utilização de corticoides tem sido favorável no controle inflamatório, principalmente naqueles casos em que antes da gravidez se fazia uso de imunossupressores. Casuística: Foi relatado um caso de uma gestante portadora de AT, através da análise de prontuário e de exames complementares, sendo realizado o pré-natal pelas equipes de Obstetrícia e Reumatologia, onde foi realizado tratamento clínico com corticoides, mostrando uma evolução satisfatória, ocorrendo apenas uma hospitalização que foi seguida de uma cesárea na trigésima sexta semana de gravidez, com recém nato saudável de 3.810g. Discussão: A AT pode estar associada a várias etiologias, sendo a gênese pouco conhecida. O diagnóstico na maioria das vezes é demorado, pela dificuldade da suspeita clínica, além de demandar o uso de técnicas de imagem mais sofisticadas. A gestação associada é fenômeno raro, já que as portadoras são orientadas a evitarem a gravidez. O controle clínico permitiu uma gestação sem grandes complicações e serviu como meio para levar a gestação até praticamente o fim do terceiro trimestre. Conclusão: Deve-se ressaltar o papel do acompanhamento clínico, especialmente com esta pan-arterite, mostrando os medicamentos que podem ser utilizados nesse grupo, especialmente quando se usava imunossupressores antes da gravidez. Palavras chave: Arterite de Takayasu, Gravidez, Vasculite. ABSTRACT:Introduction: Takayasu's arteritis (TA) consists of a vascular disease of unknown origin and chronic nature, which affects the aorta and its main branches. In pregnant women it is a complex condition, in which the clinical treatment is performed with restrictions. The use of corticosteroids has been favorable to control inflammation, especially in those cases when immunosuppressant had been used before pregnancy. Case Report: A case of a pregnant patient with TA has been reported through the analysis of medical records and laboratory tests. The Obstetrics and Rheumatology staff performed the prenatal care, and clinical treatment with corticosteroids was done showing a satisfactory outcome. There was only one hospitalization of the patient, which was followed by a cesarean in the thirty sixth week of pregnancy.  The newborn was a healthy one, weighing 3.810g. Discussion: TA may be associated with several etiologies, and the genesis is little known. The diagnosis most often takes time due to the difficulty of clinical suspicion, and requires the use of more sophisticated imaging techniques. The pregnancy is rare phenomenon, since the carriers are advised to avoid pregnancy. The clinical management allowed a pregnancy without major complications and served as a means to carry the pregnancy to almost the end of the third quarter. Conclusion: The role of the clinical follow-up should be noticed especially with this pan-arteritis, showing the drugs that can be used in this group, especially when there is use of immunosuppressive drugs before pregnancy. Keywords: Takayasu Arteritis, Pregnancy, Vasculitis.


2012 ◽  
Vol 2 (1) ◽  
pp. 16
Author(s):  
Takeshi Kusunoki ◽  
Katsuhisa Ikeda

We report a case of a neuroendocrine carcinoma arising in a wound of the postoperative maxillary sinus that was difficult to distinguish from a postoperative maxillary cyst. The patient was a 65-year-old Japanese woman who complained of left exophthalmos with cheek swelling and eye movement disorders. In past history, she had, 40 years previously undergone operation on the bilateral maxillary sinus by Caldwell-Luc’s method. In a preoperative computed tomography, a mass occupied the left maxillary sinus showing irregular densities with destruction of the posterior bone walls and invasion into the left orbital. Both TI and T2 weighted magnetic resonance imaging showed low intensities and unevenness in the mass. We performed a biopsy of the maxillary tumor according to Caldwell-Luc’s method. Histological examination diagnosed neuroendocrine carcinoma. Radiation therapy (total 66Gy) resulted in partial response for this tumor. However, sinonasal neuroendocrine carcinoma has been identified as highly aggressive, with a high probability of recurrence and metastasis.


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